To the Editor: Diagnosing celiac disease is relatively easyin typical cases, characterized by the classic features of chronicdiarrhea, abdominal pain and distention, and weight loss. However,most patients have atypical celiac disease, with few or no gastrointestinalsymptoms and a predominance of extraintestinal features (e.g.,iron-deficiency anemia). Patients with celiac disease are proneto the development of long-term complications (e.g., autoimmunediseases or cancers) that are responsible for a mortality ratehigher than that in the general population.1 Gluten withdrawalseems to be protective against long-term complications, andit is the cornerstone of treatment for celiac disease. Casefinding in subjects with clinical conditions known to be associatedwith celiac disease is currently the best epidemiologic approachto detecting atypical cases2; highly sensitive and specificserologic tests (for endomysial antibodies and tissue transglutaminaseantibodies) are now available to screen at-risk patients. Althoughthe natural history of celiac disease detected on screeningremains unclear, at present, a gluten-free diet is deemed mandatoryeven for patients with atypical celiac disease; consequently,discovering the submerged part of the "celiac iceberg" is ofthe utmost importance.
A 33-year-old man presented at our clinic with a 4-month historyof an erythematous, atrophic area on the dorsum of the tongue,accompanied by a painful burning sensation (Figure 1). He wasotherwise well and was taking no medications. His medical historywas notable only for occasional episodes of diarrhea, for whichthe patient had never sought medical advice. Results of laboratorytests, including a complete blood count and measurements ofserum iron, ferritin, vitamin B12, and folic acid levels, werewithin the normal limits. A test for endomysial antibodies waspositive. Biopsy of the small intestine showed intraepitheliallymphocytosis, crypt hypertrophy, and reduction in villous height,which confirmed the diagnosis of celiac disease. The glossitisresolved within 1 month after the initiation of a gluten-freediet.
Figure 1. Erythematous, Atrophic Area on the Dorsum of the Tongue.
The tongue was the most frequently affected site in a seriesof 128 patients with celiac disease who were examined for oralmucosal lesions and symptoms, with 29.6% of the patients describingsoreness or a burning sensation and 8.6% having erythema oratrophy.3 Robust evidence in support of routine screening forceliac disease in patients with atrophic lesions of the tongueis lacking. Nevertheless, the National Institutes of Healthconsensus statement on celiac disease states that "the singlemost important step in diagnosing celiac disease is to firstconsider the disorder by recognizing its myriad clinical features."4Our report should alert physicians and dental practitionersto consider celiac disease in managing cases of idiopathic atrophicglossitis.
Luca Pastore, D.D.S. Lorenzo Lo Muzio, M.D., Ph.D. University of Foggia 71100 Foggia, Italy
Rosario Serpico, M.D. Second University of Naples 80138 Naples, Italy
References
Corrao G, Corazza GR, Bagnardi V, et al. Mortality in patients with coeliac disease and their relatives: a cohort study. Lancet 2001;358:356-361. [CrossRef][Web of Science][Medline]
Fasano A. European and North American populations should be screened for coeliac disease. Gut 2003;52:168-169. [Free Full Text]
Lahteenoja H, Toivanen A, Viander M, et al. Oral mucosal changes in coeliac patients on a gluten-free diet. Eur J Oral Sci 1998;106:899-906. [CrossRef][Web of Science][Medline]
NIH consensus statement on celiac disease. NIH Consens State Sci Statements 2004;21:1-23. [Medline]
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